Humphrey Ayla, Higgins J Nicholas P, Yates John R W, Bolton Patrick F
Developmental Psychiatry Section, University of Cambridge, Addenbrooke's Hospital, Cambridge, Institute of Psychiatry, London, UK.
Neurology. 2004 Mar 9;62(5):795-8. doi: 10.1212/01.wnl.0000113745.58425.ef.
A pair of monozygotic male twins with tuberous sclerosis (TS) were followed between 18 months and 3 years of age. Twin A with 25 large cortical tubers and hence extensive brain involvement was moderately mentally retarded and met criteria for autism. The other twin had more (n = 31) but smaller tubers. He was not mentally retarded and did not meet criteria for autism. This study provides evidence that nongenetic factors such as extent of brain abnormality and not just number of cortical tubers are important in determining phenotypic variability in TS. The findings also raise questions about the mechanisms giving rise to autism in TS.
一对患有结节性硬化症(TS)的单卵双胞胎男性在18个月至3岁期间接受了跟踪观察。双胞胎A有25个大的皮质结节,因此脑部受累广泛,有中度智力障碍,符合自闭症标准。另一个双胞胎有更多(n = 31个)但更小的结节。他没有智力障碍,也不符合自闭症标准。这项研究提供了证据,表明非遗传因素如脑异常程度而非仅仅皮质结节数量在决定TS的表型变异中很重要。这些发现也引发了关于TS中导致自闭症的机制的问题。